Methotrexate‑related other iatrogenic immunodeficiency‑associated lymphoproliferative disorder in the CNS and medication‑related osteonecrosis of the jaw occurring simultaneously: A case report

Kato,Tomoki; Mizumoto,Chisaki; Inoue,Fuminori; Watanabe,Takuma; Yamanaka,Shigeki; Fukuhara,Shizuko; Nakao,Kazumasa

doi:10.3892/etm.2023.12329

Methotrexate‑related other iatrogenic immunodeficiency‑associated lymphoproliferative disorder in the CNS and medication‑related osteonecrosis of the jaw occurring simultaneously: A case report

Authors:
- Tomoki Kato
- Chisaki Mizumoto
- Fuminori Inoue
- Takuma Watanabe
- Shigeki Yamanaka
- Shizuko Fukuhara
- Kazumasa Nakao
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Affiliations: Department of Oral and Maxillofacial Surgery, Graduate School of Medicine, Kyoto University, Kyoto 606‑8507, Japan, Department of Hematology and Oncology, Graduate School of Medicine, Kyoto University, Kyoto 606‑8507, Japan
Published online on: November 28, 2023 https://doi.org/10.3892/etm.2023.12329
Article Number: 41
Copyright: © Kato et al. This is an open access article distributed under the terms of Creative Commons Attribution License.

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Abstract

Methotrexate‑related other iatrogenic immunodeficiency‑associated lymphoproliferative disorder (MTX‑OIIA‑LPD) is prone to extranodal involvement but rarely involves the central nervous system (CNS). The present study reports a case of MTX‑OIIA‑LPD of the CNS discovered during medication‑related osteonecrosis of the jaw (MRONJ) treatment in a 76‑year‑old woman with rheumatoid arthritis (RA). The chief complaint of the patient was bone exposure and pain in the right mandibular molar. The patient had been receiving MTX for RA and alendronate sodium hydrate for osteoporosis, followed by denosumab. Treatment was initiated based on a diagnosis of MRONJ. However, the patient experienced lightheadedness and floating dizziness afterwards. Examinations revealed scattered neoplastic lesions in the brain. The histopathological diagnosis was diffuse large B‑cell lymphoma. A systemic search also revealed adrenal involvement. Since the patient was taking MTX, a diagnosis of MTX‑OIIA‑LPD was made and MTX was discontinued. Chemotherapeutic agents were administered since the central lesions became symptomatic. The MTX‑OIIA‑LPD lesions in the brain and adrenal glands completely resolved 8 months after onset. The physical condition of the patient improved, and the bone‑exposed areas became epithelialized. Reports on MTX‑LPD in the oral and maxillofacial region are few, which may delay its diagnosis. Therefore, biopsy of oral lesions in patients with MRONJ who are taking MTX and collaboration with related diagnostic departments, such as rheumatology and hematology, must be done to initiate the diagnosis and treatment of extraoral MTX‑LPD.

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